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Home , Eumycetoma, Madurella mycetomatis, Mycetoma

Karrakchou et al. BMC Dermatology (2020) 20:1 https://doi.org/10.1186/s12895-019-0097-1

CASE REPORT Open Access mycetomatis of the foot: a case report of a neglected tropical disease in a non-endemic region Basma Karrakchou* , Ibtissam Boubnane, Karima Senouci and Badreddine Hassam

Abstract Background: is an uncommon chronic granulomatous infection of cutaneous and subcutaneous tissues that can be caused by filamentous bacteria (actinomycetoma) or fungi (). It is the prerogative of young men between the third and fourth decade and is transmitted through any trauma causing an inoculating point. The classic clinical triad associates a painless hard and swelling subcutaneous mass, multiple fistulas, and the pathognomonic discharge of grains. Although endemic in many tropical and subtropical countries, mycetoma can also be found in non-endemic areas as in Morocco, and causes then diagnosis problems leading to long lasting complications. Therefore, we should raise awareness of this neglected disease for an earlier management. Under medical treatment however, mycetoma has a slow healing and surgery is often needed, and relapses are possible. Case presentation: Herein we report a case of a 64 years old patient, with a history of eumycetoma occurring ten years ago treated with oral coupled with surgery. A complete remission was seen after 2 years. He presented a relapse on the previous scar 6 months ago. There wasn’t any bone involvement in the magnetic resonance imaging (MRI). The patient was put under oral terbinafine with a slow but positive outcome. Conclusion: Through this case report, we perform a literature review and highlight the importance of increase awareness of mycetoma in clinical practice especially in non-endemic regions. Keywords: , Mycetoma, Actinomycetoma, Eumycetoma, Neglected disease

Background regions with low rainfall [3, 6]. Therefore, mycetoma is en- Mycetoma is a chronic granulomatous infection of cutane- demic in many countries in the tropics and subtropics [3], ous and subcutaneous tissues that can be caused by fila- but little known in other countries including Morocco mentous bacteria (actinomycetoma) or fungi (eumycetoma) where they prevail in sporadic forms and cause diagnostic [1]. It occurs typically in young men between 20 and 40 problems [7]. years old, and is the prerogative of farmers who are ex- In this work, a case of eumycetoma of the foot in a posed to contaminated soil during minor injuries in most Moroccan patient was reported, and literature review cases [2]. Clinically, mycetoma or « Madura foot » is an was provided. inflammatory tumor, often polyfistulised, evolving in a chronic mode. Its fistulas give rise to grains whose color di- Case presentation rects towards the causative germ [3, 4]. Although the true Our patient is a 64 years old man from Sidi Slimane in incidence of mycetoma remains uncertain, 60% of myce- the center of Morocco where he currently lives and toma are bacterial and 40% are fungal [5]. And most of the works as a farmer. He is married and has five children in cases fall between latitude 15°S and 30°N, the so-called good health. He presented ten years ago a history of a “mycetoma belt” characterized by warm, dry, semi-desert located in the dorsal surface of his right forefoot, which has progressively increased in size until becoming a swelling and slightly painful polyfistulised tumor emit- * Correspondence: [email protected] Dermatology and Venereology Department, Ibn Sina Hospital, Mohammed V ting dark grains. No previous trauma or injury of the University, Rabat, Morocco foot has been noticed by the patient and no travel to

© The Author(s). 2020 Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated. Karrakchou et al. BMC Dermatology (2020) 20:1 Page 2 of 7

endemic zone in Africa or outside Africa has been re- On the other hand, several differential diagnoses of myce- ported. He was hospitalized in the Dermatology depart- toma such as cutaneous , profound , fis- ment of the University Hospital Ibn Sina of Rabat, and tulised osteitis, leprosy, and cutaneous metastatic lesions the diagnosis of eumycetoma due to Madurella myceto- were considered. Complementary examinations showed a matis was detected. The patient was initially treated with normal aspect on foot X-rays. The biological assessment oral at a dosage of 400 mg/day during 2 didn’t show any signs of bacterial infection. A was years. After treatment, the lesions did not im- performed and during this procedure, a serohematic dis- prove substantially and itraconazole was substituted by charge was witnessed with conglomerates of small and firm terbinafine 500 mg/day, associated with surgical debride- blackish pellets, evoking eumycetoma (Fig. 1). Tissue and ment of the tumor. The outcome was good with total black grain samples were analyzed for bacterial, mycological healing of the lesions, and treatment with terbinafine and histological evaluation. The anatomopathological as- was continued to achieve a total length of 6 months. pect showed a polymorphic inflammatory cell reaction. The The current history goes back to 6 months by the bacterial analysis was negative, and the mycological study appearance of a nodule localized on the previous scar, revealed on direct examination septated hyphae containing having the same evolution than the previous one. On numerous chlamydoconidia measuring 2 to 5 μmindiam- physical examination, he had an indurated inflammatory eter with terminal dilatations giving a vesicle appearance tumor of the dorsal surface of the right forefoot, measur- (Fig. 2). The fungal culture on Sabouraud media established ing 15x10cm, adherent to the and to deep structures, the diagnosis of certainty and identified Madurella myceto- with many visible openings, which let emerge seropuru- matis after 3 weeks of growth (Fig. 3). A magnetic reson- lent sometimes hematic fluid and small black grains of 1 ance imaging of the right foot was performed to determine to 2 mm (Fig. 1). Moreover, inflammatory inguinal lymph- the lesions extension. This examination identified multiple adenopathy was found in the right side, and macerated collections of the right forefoot soft tissues, fusing along ex- toes intertrigos were present in the right foot. Further- tensor and flexor tendons without associated joint or bone more, there was no fever or alteration of the general invasion (Fig. 4). A treatment with oral terbinafine at a dos- condition. age of 750 mg/day for at least 1 year was started because of its previous efficiency in our patient. We performed monthly liver function tests (liver transaminases) to assess treatment tolerability. Within 6 months, the lesions evolu- tion was slow with a fistulas drying up, and no liver damage was noted.

Fig. 2 Direct examination of black grains under optical microscopy × 40 (Parasitology and Mycology Department, Ibn Sina Hospital, Rabat). Evocative aspect of Madurella mycetomatis: thick septated Fig. 1 Mycetoma of the right forefoot with polyfistulas emitting and branched hyphae (red arrow), ending in vesicles corresponding serohematic fluid and dark grains (arrow) to circular chlamydoconidia (black arrow) Karrakchou et al. BMC Dermatology (2020) 20:1 Page 3 of 7

intermittent short periods of rainfall. These zones are called “mycetoma belt” and they include Mauritania, Senegal, Chad, Sudan, Ethiopia, Somalia, Yemen, India, Thailand, Mexico, and the Bolivarian Republic of Venezuela. Therefore the World Health Organization approved (May 2016) a resolution (WHA69.21) recog- nizing mycetoma as a neglected tropical disease because of its endemicity in poor populations living in remote areas of developing countries [1]. However, some spor- adic autochthone cases have been described in temper- ate zones such as in the Maghreb, where the disease incidence is rare, as in Morocco, causing diagnosis problems. Mycetoma is the prerogative of young adults, mostly men between the third and fourth decade with a sex ratio of 4/1 [5]. The transmission is done via a contact with a Fig. 3 Microscopic aspect of Madurella mycetomatis’ colony colored contaminated soil through a thorn prick or any trauma with Lactophenol Cotton Blue Stain × 40 (Parasitology and Mycology causing an inoculating point [1]. However, the infection is Department, Ibn Sina Hospital, Rabat) not transmitted from one person to another. Therefore, mycetoma is found generally in rural areas and affects manual workers or those who walk barefoot, such as Discussion and conclusions farmers, laborers, and herdsmen. Furthermore, our patient Mycetoma was first reported in 1843 by Dr. John Gill in corresponds to the description of the persons at risk to de- Madurai, India [8] and was then called “Madura foot”.It velop the disease in view of his sex, origin, and work. is a chronic granulomatous infection of cutaneous and The classic clinical triad associating a painless hard subcutaneous tissues that can be caused by filamentous and swelling subcutaneous mass, multiple fistulas, and bacteria (actinomycetoma) or fungi (eumycetoma) [1]. discharge of grains characterizes mycetoma. The foot is Indeed, a bacterial origin is found in 60%, whereas fungi the most common site of involvement, as seen in our pa- are responsible of 40% of cases reported worldwide [2]. tient and as indicated by the denomination “Madura Moreover, causative bacterial agents are more numerous foot” [10]. Nonetheless, all other parts of the body can than fungal agents (only four agents reported) [9] be affected such as the arm, forearm, hand, back, thorax, (Table 1). The mycetoma’s agents are found throughout head and neck [11]. The physiopathological steps are the world, while they are endemic in the tropical and characterized by four phases. A first long period of incu- subtropical zones with hot and dry climates, and bation characterizes this disease, going from a few weeks

Fig. 4 Magnetic resonance imaging scan of the right foot showing the « dot in circle » sign (arrow). a-Sagittal view; b-Transversal view Karrakchou et al. BMC Dermatology (2020) 20:1 Page 4 of 7

Table 1 Clinical, radiological and microbiological differential features between eumycetoma and actinomycetoma Eumycetoma Actinomycetoma Causative organism =Fungi, mainly four =Bacteria, numerous Madurella mycetomatis (most common) Nocardia spp. (mostly in regions with higher humidity) Madurella grisea Nocardia brasiliensis Pseudoallescheria boydii Nocardia asteroides Leptosphaeria senegalensis Nocardia otidiscaviarum Others … Actinomadura spp Actinomadura madurae Actinomadura pelletieri somaliensis Actinomyces israeli Others … Clinical Slow evolution Rapid evolution Most often the foot Most commonly the foot, but also the chest, head and abdomen Well-limited tumor with a clear margin Diffuse mass with no clear margin, more inflammatory and White or black grains destructive Rare lymphatic metastasis Many grain colors, but not black Frequent lymphatic metastasis MRI Few but large well limited soft tissues cavities Numerous soft tissues cavities with a small size and unclear margins Less osteophilic Rapid bone involvement Direct microscopic Black or white Red or white examination of the grains Diameter > 3 μm Diameter < 1 μm Few thick hyphae dilated in places to form Thin and numerous grains, no hyphae vesicles Fringes in the outskirt No fringes Gram stain Periodic-acid–Schiff stain Gomori methanamine silver stain Culture of the grains Sabouraud - culture media Sabourauld without antibiotics media Slow growth 2–3 weeks Loewenstein culture media Rapid growth to several years (6 months in our patient). Then appears toward the causative germ (Table 2). The classic Madura a discomfort and pain, followed by the constitution of a tumor is then constituted and usually is superinfected slightly inflammatory subcutaneous nodule of 1 to 4 cm leading sometimes to fatal sepsis. Left untreated, the in- of diameter, gradually increasing in size. At the state fection spreads through deep structures including fascia phase, the infection extends to superficial structures, and planes and the underlying bone and muscle. Lymphatic fistulises to the skin, leaving grains whose color directs extension has been reported in few cases, as in our pa- tient who presented with right inguinal lymphadenop- Table 2 Main mycetoma agents depending on the color of the athy [12]. It is to highlight that the clinical aspects of grains eumycetoma and actinomycetoma are almost similar, Dark grains Madurella mycetomatis (Sahelian with few differences (Table 1). =fungi mycetoma Africa, Middle East, India) Differential diagnoses of mycetoma mainly include Madurella grisea (South America) other subcutaneous with similar presentations. Leptosphaeria senegalensis (Mauritania, Senegal) Cutaneous tuberculosis, especially gumma of the foot, Leptosphaeria tompkinsii (Exceptional) represents the main in our context, Pyrenochaeta reomeroi (Rare) as Morocco remains an endemic country where tubercu- jeanselni (Rare) losis still causes ravages. Other atypical mycobacteriosis, Red grains Actinomadura pelletieri (West Africa) , soft tissue tumors, and chronic fistulised =bacterial mycetoma osteomyelitis can have the same clinical presentation but White and yellow grains Actinomadura madurae (Cosmopolitan) the pathognomonic presence of grains rectifies the diag- Streptomyces somalienis (Desert and subdesertic regions) nosis [13]. Recently, dermoscopy has proven its useful- Nocardia spp (Tropical humid regions) ness in detecting subclinical grains. It gives a clinical White grains Pseudallescheria budii (Quite rare, tropical diagnosis presumption by showing structureless blue- and temperate regions) white areas in eumycetoma, corresponding to deep black spp (Rare) spp (Rare) grain localization. A white halo surrounds these areas, Neotestudina rosatii (Exceptional) and sometimes polymorphic vessels are seen [14]. spp (Exceptional) The mycetoma diagnosis is primarily clinical. Additional (Rare) tests are performed, on the one hand, to determine the Karrakchou et al. BMC Dermatology (2020) 20:1 Page 5 of 7

causative organism for appropriate treatment, and on the Morocco. Their use would have decreased the required other hand to look for the tumor extension. Direct micro- time for the species diagnosis but has no impact on the scopic examination of the grains can already give an etio- therapeutic choice in this case. Indeed, the black color of logical orientation. The grains are collected with a scalpel the grains on direct examination directs towards six pos- and placed between blade and lamella with the KOH solu- sible fungal species (Table 2), and can already give a thera- tion. The size, color, consistency of grains, and the exist- peutic orientation. It has been opted in our case for ence of cement makes it possible to distinguish fungal classical methods for species diagnosis (direct examination grains from actinomycotic ones (Table 1). Actinomycotic and grain culture) considering their low cost and accessibil- grains are red or white elements with a diameter < 1 μm, ity, and their sensitivity and specificity. However, in en- thin and numerous, with fringes in the outskirt. The fungal demic areas where diagnostic possibilities are limited, ones are black measuring > 3 μm of diameter, with few culture is rarely done and then the color and the macro- thick hyphae dilated in places to form vesicles with no scopic appearance of grains are used. The anatomopatholo- fringes, as seen in our patient [1](Fig.2). For the species gical examination is particularly indicated when the patient diagnosis, the grains are seeded and deposited in tube cul- is seen at a non-productive fistulas stage. Hematein-eosin ture media on Sabouraud-antibiotics culture media without staining is generally sufficient to study the grains, but de- actidione if they are fungal, or on Sabouraud without anti- pending on their appearance, specific staining may be re- biotics or Loewenstein culture media if they are actinomy- quired, and are helpful in differentiating organisms cotic. Depending on the species, the cultures are more or histopathologically: Periodic-acid–Schiff or Gomori- less rapid to obtain. Species diagnosis is based on the Grocott for fungi, Gram for actinomycetes. The reaction macroscopic appearance of the cultures and microscopy around the grains is generally granulomatous with granulo- (Fig. 3). In our patient, Madurella mycetomatis specific cytes in contact with the grains, all surrounded by histio- characteristics were identified on direct examination of cytes and lymphocytes with neo-vessels. A third layer grain and its culture. The grains are macroscopically black consists of fibrosis [17]. In our patient, the histopathology to brown firm grains from 0.5 to 1 mm. Direct microscopic revealed the polymorphous inflammation. Moreover, med- examination is evocative and shows 3–4 μm septated hy- ical imaging is particularly interesting in the assessment of phae branched in a network and ended in circular vesicles mycetoma extension especially bone involvement, which is (chlamydoconidia) (Fig. 2). Madurella mycetomatis culture more frequent in actinomycetoma due to it being more grows slowly after at least 3 weeks at 27 °C on Sabouraud osteophilic than fungal agents. Without treatment, this medium with antibiotics and without actidione. The culture complication leads to functional impotence and can lead to macroscopic aspect is a circular flat felting colony with an amputation. Therefore standard radiography is compulsory elevated center and peripheral grey folds. The reverse side and can show a cortical thinning or hypertrophy, bone cav- is dark to brown with a diffusible pigment in agar. The cul- ities, and osteoporosis, which we didn’tfindinourpatient ture microscopic examination with Lactophenol Cotton who had a fungal infection [18]. Ultrasonography is helpful Blue Stain shows septated blue hyphae and chlamydoconi- for soft tissue extension. However, magnetic resonance dia (Fig. 3). The absence of MALDI-TOF and PCR analysis imaging remains the gold standard to assess both the soft are major limitations of the present report. The develop- tissues and early bone involvements. Indeed, MRI shows ment of soft ionization techniques for mass spectrometry the “dot-in-circle” sign (Fig. 4), corresponding to a central such as MALDI-TOF (Matrix-Assisted Laser Desorption spherical hypointense signal (grains), surrounded by a Ionization Time-Of-Flight) allows a large panel analysis of hyperintense signal (the granuloma), with a peripheral low- specific species biomarkers. MALDI-TOF is more accurate signal matrix representing fibrous tissue [18]. Likewise, we than conventional phenotypic techniques in species identifi- found this sign in our patient as seen in Fig. 4,moreover, cation with a lower cost per identification and a faster re- there was no bone involvement then no bone hypersignal. sult.Indeed,MALDI-TOFisperformed directly on samples The mycetoma treatment must be started as soon as without prior culture, which is useful for non-cultivable or possible before an advanced stage when amputation is slow-growing microorganisms [15]. Fungal DNA detection the only therapeutic option. For that, there are much by PCR (Polymerase Chain Reaction)isalsoatechnique more therapeutic molecules to target actinomycetoma used for species diagnosis. Molecular biology is performed germs than eumycetoma ones. Moreover, eumycetoma on samples without a former culture. Unfortunately, it is usually refractory to medications and an extended doesn’t always discriminate species, and other targeted duration of treatment for at least 1 year is needed, genes are required. In addition to the high cost, this tech- against 3 months for bacterial mycetoma [19]. Indeed, nique requires high expertise and should be reserved for we noticed in the first episode of mycetoma (ten years non-cultivable microorganisms [16]. Moreover, MALDI- ago) in our patient a necessary duration of two years TOF and PCR were not used in our patient because of their and a half for total healing of the fungal lesions. More- lack of availability at the University Hospital of Rabat, over, for the current history, there is a slow Karrakchou et al. BMC Dermatology (2020) 20:1 Page 6 of 7

improvement of his eumycetoma after 6 months of treat- Ethics approval and consent to participate ment. The molecules available for eumycetoma treat- Not applicable. ment include imidazoles such as (400 mg/ day), itraconazole (200–400 mg/day), (200 Consent for publication Written informed consent was obtained from the patient for publication of mg/day), (400–600 mg/day); amphotericin this case report and accompanying images. B (0.5–1.25 mg/kg per day); and terbinafine (500–1000 mg/day), alone or in any combination [20]. For our pa- Competing interests tient, oral administration of terbinafine at a dosage of The authors declare that they have no competing interests.

750 mg/day was adopted because of its previous effi- Received: 1 July 2019 Accepted: 19 December 2019 ciency, availability and low cost comparing to other mol- ecules. For actinomycetoma, trimethoprim- sulfamethoxazole is the most effective [21]. References On the other hand, surgical tumorectomy is performed 1. Zijlstra EE, van de Sande WWJ, Welsh O, Mahgoub ES, Goodfellow M, Fahal AH. Mycetoma: a unique neglected tropical disease. Lancet Infect Dis. 2016; in small, localized lesions, and also for large lesions to 16(1):100–12. reduce the mass size for better medical treatment effect- 2. Sampaio FM, Galhardo MC, Quintella LP, Souza PR, Coelho JM, Valle AC. iveness [22]. Indeed, relapses can occur and a follow-up Eumycetoma by Madurella mycetomatis with 30 years of evolution: a therapeutic challenge. An Bras Dermatol. 2013;88(6 Suppl 1):82–4. for many years is needed to ensure complete remission 3. Ahmed AO, Van Leeuwen W, Fahal A, Van de Sande W, Verbrugh H, Van of the disease. 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